An adult with cystathionine beta-synthase deficiency, camptodactyly-arthropathy-coxa vara-pericarditis syndrome, and deafness: A case report.
Karina Carvalho DonisMarco Antônio Baptista KalilFabiano de Oliveira PoswarFernando KokCharles Lubianca KohemSoraia PoloniTaciane BorsattoFilippo Pinto E VairoFranciele Cabral PinheiroIda Vanessa Döederlein SchwartzPublished in: Genetics and molecular biology (2024)
Massive sequencing platforms allow the identification of complex clinical phenotypes involving more than one autosomal recessive disorder. In this study, we report on an adult patient, born to a related couple (third degree cousins), referred for genetic evaluation due to ectopia lentis, deafness and previous diagnosis of juvenile idiopathic arthritis. He was biochemically diagnosed as having Classic Homocystinuria (HCU); Sanger sequencing of the CBS gene showed the genotype NM_000071.2(CBS):c.[833T>C];[833T>C], compatible with the diagnosis of pyridoxine-responsive HCU. As he also had symptoms not usually associated with HCU, exome sequencing was performed. In addition to the variants found in the Sanger sequencing, the following variants were identified: NM_001256317.1(TMPRSS3):c.[413C>A];[413C>A]; and the NM_005807.6(PRG4):c.[3756dup]:[3756dup], confirming the diagnosis of autosomal recessive nonsyndromic deafness and Camptodactyly-Arthropathy-Coxa Vara-Pericarditis Syndrome (CACP), respectively. Genomic analysis allowed the refinement of the diagnosis of a complex case and improvement of the patient's treatment.
Keyphrases
- copy number
- case report
- single cell
- juvenile idiopathic arthritis
- photodynamic therapy
- genome wide
- intellectual disability
- gene expression
- rheumatoid arthritis
- cancer therapy
- drug delivery
- physical activity
- transcription factor
- low birth weight
- disease activity
- duchenne muscular dystrophy
- gestational age
- muscular dystrophy
- preterm birth